[CIS PIDD] [cis-pidd] Laryngeal papilomatosis in boy with CGD awaiting BMT

Sun Mar 12 17:22:02 EDT 2017

The thought is that kids with HPV+ papilomatosis acquire it through the
birth canal. Another good reason to vaccinate.
Michael Weinreich

Michael Weinreich

On Sun, Mar 12, 2017 at 5:13 PM, CIS-PIDD <cis-pidd at lists.clinimmsoc.org>
wrote:

> Am I the only person wondering if child protection should be involved in
> oral HPV in a 9 yo boy?
>
> Kathy Haines
>
> -----Original Message-----
> From: CIS-PIDD [mailto:cis-pidd at lists.clinimmsoc.org]
> Sent: Saturday, March 11, 2017 7:45 PM
> To: CIS-PIDD <cis-pidd at lyris.dundee.net>
> Cc: jaime inostroza <jaimel.inostroza at gmail.com>
> Subject: Re: [cis-pidd] Laryngeal papilomatosis in boy with CGD awaiting
> BMT
>
> If the sender shows as an internal Hackensack Meridian email address
> forward the email to Information.Security.Office at HackensackMeridian.org
>
> Note, THIS IS AN EXTERNAL EMAIL. It did not originate at Hackensack
> Meridian Health Network.
>
> ----------------------------------------------------------------------
>
>
> Sent from my iPhone
>
> > On Mar 11, 2017, at 7:23 PM, CIS-PIDD <cis-pidd at lists.clinimmsoc.org>
> wrote:
> >
> > Dear Jane
> >
> > I am writing on behalf of Dr. Jaime Inostroza,  director of the Jeffrey
> Modell Center for Diagnosis  and Research in primary immunodeficiencies in
> Temuco, Chile. I mentioned your question to him because I was aware of an
> upcoming publication by his team.
> >
> > They evaluated  a 7-yer-old boy with chronic recurrent laryngeal
> papilomatosis requiring multiple surgeries since infancy. No
> immunodeficiency was identified. A biopsy of his lesions confirmed the
> presence of HPV serotype 6. Based on several publications about the use of
> the tetravalent HPV vaccine at 9 years of age this boy received 3 doses of
> Gardasil. The second and third doses were given at 6 mo intervals. His
> papilomatosis has cleared completely for the first time in his life. He
> leads now a normal life and has remained free of recurrences  for over 1
> year. This patient case will be published now in the Chilean Journal of
> Pediatrics. I am requesting the English abstract and references in case you
> are interested.
> >
> > CGD patients have normal antibody and cell mediated immunity and usually
> have normal responses to vaccines. You could consider the therapeutic use
> of HPV for your patient.
> >
> > Ricardo Sorensen, M.D.
> > Clinical Professor of Pediatrics
> > LDU Health Science Center and Children’s Hospital, New Orleans Head,
> > Jeffrey Modell Center for Diagnosis and Research in New Orleans
> > Honorary Professor, Universidad de la Frontera. Temuco, Chile
> >
> > -----Original Message-----
> > From: CIS-PIDD [mailto:cis-pidd at lists.clinimmsoc.org]
> > Sent: Saturday, March 11, 2017 5:39 AM
> > To: CIS-PIDD
> > Subject: RE:[cis-pidd] Laryngeal papilomatosis in boy with CGD
> > awaiting BMT
> >
> > *EXTERNAL EMAIL: EVALUATE*
> >
> >
> > Dr. Peake:
> >
> > We've had some decent experience with nebulized cidofovir.   See Giles
> et al., Ped Resp Rev 2006.
> >
> > The more curious question is why an XL-CGD patient would have --
> assuming it's confirmed as HPV positive -- a recurrent viral infection.
> >
> > Good luck with this case.
> >
> >   - K
> >
> > Karl O. A. Yu, M.D., Ph.D., F.A.A.P.
> > Section of Infectious Diseases  |  Department of Pediatrics  |  Comer
> > Children's Hospital  |  University of Chicago
> > 5841 S Maryland Ave,  MC 6054,  Chicago  IL  60637 Office phone:
> > 773-702-9281  |  Pager: 773-702-6800 x1744  |  Fax: 773-702-1196
> >
> > ________________________________________
> > From: CIS-PIDD [cis-pidd at lists.clinimmsoc.org]
> > Sent: Thursday, March 09, 2017 5:40 AM
> > To: CIS-PIDD
> > Subject: [cis-pidd] Laryngeal papilomatosis in boy with CGD awaiting
> > BMT
> >
> > Dear all
> > I was wondering if anyone could help with a patient we have?
> >
> > We have a 13yo boy with X-linked CGD (previous liver abscess, pulmonary
> aspergillosis, chronic lung disease) who we were working up for BMT. He has
> had some palatal ulcers (biopsy - granuloma; extensive microbiological
> workup revealed no organism) and multiple lymph nodes in his neck for a
> number of months.  He was admitted prior to transplant to improve nutrition
> and to investigate some potential foci of infection. In the workup for
> transplant he was noted to have on MRI asymmetric but circumferential
> thickening of the soft palate, uvula, palatine tonsils, oropharyngeal
> mucosal space, epiglottis, aryepiglottic folds, larynx and larynopharynx.
> PET scan showed diffuse moderate to intense FDG uptake in the same areas.
> On initial bronchoscopy he was noted to have some supraglottic nodules
> which were presumed to be granulomas and were not biopsied. A course of
> steroids were commenced with prednisone 1mg/kg given for 10 days then
> weaned over the following 3 weeks. He then started to get increasing
> problems with his upper airways with production of a lot of purulent mucus
> (no growth) and some difficulty swallowing. On repeat  laryngoscopy he was
> found to have diffuse severe papillomatosis of the whole of the larynx with
> copious overlying purulent exudate. Histology would be consistent with
> papillomatosis and is p16 positive and we are awaiting PCR for HPV. He
> responded well initially to debridement and relatively high dose
> dexamethasone (he already was covered with broad spectrum antibiotics and
> ambisome). Due to a rapid return of his supraglottic and glottic swelling
> which followed weaning of his steroids and cessation of antibiotics (no
> bacterial growth), he is to have further debridement tomorrow just 2 weeks
> after this was first undertaken.
> >
> > Has anyone encountered HPV this in this setting? If so, what treatments
> have you tried? What about BMT with  laryngeal papillomatosis ?
> >
> > Any help or suggestions greatly appreciated
> >
> > Kind regards
> >
> > Jane
> >
> > Associate Professor Jane Peake
> > Paediatric Immunologist and Allergist
> > Discipline of Paediatrics & Child Health School of Medicine | The
> > University of Queensland Lady Cilento Children's Hospital| 501 Stanley
> > Street | South Brisbane QLD 4101
> >
> >
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